Trends of cleft surgeries and predictors of late primary surgery among children with cleft lip and palate at the University College Hospital, Nigeria: A retrospective cohort study

Background Cleft of the lip and palate is the most common craniofacial birth defect with a worldwide incidence of one in 700 live births. Early surgical repairs are aimed at improving appearance, speech, hearing, psychosocial development and avoiding impediments to social integration. Many interventions including the Smile Train partner model have been introduced to identify and perform prompt surgical procedures for the affected babies. However, little is known about the trends of the incidence and surgical procedures performed at our hospital. Nothing is also known about the relationship between the clinical characteristics of the patients and the timing of primary repairs. Objective To determine the trends in cleft surgeries, patterns of cleft surgeries and identify factors related to late primary repair at the University College Hospital, UCH, Ibadan, Nigeria. Methods A retrospective cohort study and trends analysis of babies managed for cleft lip and palate from January 2007 to January 2019 at the UCH, Ibadan was conducted. The demographic and clinical characteristics were extracted from the Smile Train enabled cleft database of the hospital. The annual trends in rate of cleft surgeries (number of cleft surgeries per 100,000 live births) was represented graphically. Chi square test, Student’s t-test and Mann Whitney U were utilised to assess the association between categorical and continuous variables and delay in cleft surgery (≥12 months for lip repair, ≥18months for palatal repair). Kaplan-Meier graphs with log-rank test was used to examine the association between sociodemographic variables and the outcome (late surgery). Univariable and multivariable Cox proportional hazard regression was conducted to obtain the hazard or predictors of delayed cleft lip surgery. Stata version 17 (Statacorp, USA) statistical software was utilised for analysis. Results There were 314 cleft surgeries performed over the thirteen-year period of study. The male to female ratio was 1.2:1. The mean age of the patients was 58.08 ± 99.65 months. The median age and weight of the patients were 11 (IQR:5–65) months and 8 (IQR: 5.5–16) kg respectively. Over half (n = 184, 58.6%) of the cleft surgeries were for primary repairs of the lip and a third (n = 94, 29.9%) were surgeries for primary repairs of the palate. Millard’s rotation advancement flap was the commonest lip repair technique with Fishers repair introduced within two years into the end of the study. Bardachs two flap palatoplasty has replaced Von Langenbeck palatoplasty as the commonest method of palatal repair. The prevalence of late primary cleft lip repair was about a third of the patients having primary cleft lip surgery while the prevalence of late palatal repair was more than two thirds of those who received primary palatoplasty. Compared with children who had bilateral cleft lip, children with unilateral cleft lip had a significantly increased risk of late primary repair (Adj HR: 22.4, 955 CI: 2.59–193.70, P-value = 0.005). Conclusion There has been a change from Von Langenbeck palatoplasty to Bardachs two-flap palatoplasty. Intra-velar veloplasty and Fisher’s method of lip repair were introduced in later years. There was a higher risk of late primary repair in children with unilateral cleft lip.


Conclusion
There has been a change from Von Lagenbeck palatoplasty to Bardachs two-flap palatoplasty. Intra-velar veloplasty and Fisher's method of lip repair were introduced in later years. There was a higher risk of late primary repair in children with unilateral cleft lip.  The author(s) received no specific funding for this work.

Competing Interests
The authors have declared that no competing interests exist.

Introduction
Cleft of the lip and palate is the most common craniofacial birth defect with a worldwide incidence of one in 700 live births (1). This burden is higher in low-and middle-income countries with a higher number of untreated clefts (2). Early surgical repairs are aimed at improving appearance, speech, hearing, psychosocial development and avoiding impediments to social integration (3). Some guidelines have been put forth to optimize safety, ensure adequacy of repair, and improve function while limiting morbidity to the patient with the cleft deformity (4,5). Surgical procedures should be well planned to reduce exposure to anaesthesia from multiple surgeries (3).
One of the common guidelines for the timing of surgical cleft lip repair is the rule of ten (4).

Study design and setting
This was a retrospective cohort study of all cleft surgeries from January 2007 to December 2019.
The study was carried out at the University College Hospital, Ibadan, a tertiary care, federal Atypical clefts were clefts that involved any other part of the face that differed from the commoner pattern of cleft lip and palate. Here they were classified according to Paul Tessier (17).
Primary surgeries were defined as the first surgeries performed on any anatomical region of the cleft.
Secondary surgeries -Subsequent surgeries performed after the primary surgery on the same anatomical region of the cleft.

Variables
Primary outcome measure was Late primary surgery (age at surgery ≥ 12 months for lip repair and ≥ 18 months for palatal repair). Independent variables were age, gender, weight, type of deformity, severity of the deformity, age at surgery and year of surgery. Only primary surgeries of typical clefts were utilised for bivariate and multivariable a analysis to determine the primary outcomes, as such atypical clefts and secondary surgeries were not analyzed further.

Data analysis
Categorical

Demographic and clinical characteristics
There were 314 cleft surgeries performed over the thirteen-year period of study (January 2007-December 2019).

Trends in cleft surgeries
There

Association of demographic and clinical parameters with age at primary surgery
Nearly all the primary surgeries that were done in children less than 6

Kaplan Meir plot of the time from birth to primary surgery
The total time of follow-up was 14,770 child-month and the rate of primary cleft surgery was 18 per thousand child-months after birth. Furthermore, the median time from birth to primary surgery was 10months while 25% of the babies had surgery after 48 months. (Fig 5A, Table 4) Figure 5A. Kaplan Meir plot of the time to cleft surgeries     The gender distribution in this study is similar to other studies that have reported an overall male preponderance with female preponderance in Isolated cleft palate. (11,18,19) A study from China however observed more males in patients with isolated cleft palate, which they adduced to likely, cultural female discrimination in their setting. (12) laterality of the anomalies in our study shows a predominance of left sided clefts as reported in other studies. (18,19) The steep rise in the number of surgeries performed from the onset of the partnership could be because of the increasing awareness of free surgeries, possibly reflecting a mop-up of backlogs of unrepaired clefts. This annual low volume of cleft surgeries has earlier been reported by

Limitations of the study
The data base used for this study was limited in variables such as education of the parent s which may also have affected the late primary repair. The nature of the data base used for this research does not allow for relating the changing trend in the methods of cleft lip and palate repair to outcomes of repair.

Conclusions and Recommendations
The trends in number of cleft surgeries show a low volume in later years. There has ben a change from Von Lagenbeck palatoplasty to Bardachs two-flap palatoplasty. Intra-velar veloplasty and Fisher's method of lip repair were introduced in later years. There was a higher risk of late primary repair in children with unilateral cleft lip. There is a need to investigate the reason for the changing trends in techniques of cleft surgeries and determine the impact of this on the functional outcomes of the repairs. More attention would need to be paid to children with less severe cleft deformities to ensure they have early repairs. Increased awareness on identification of incomplete clefts of the palate may increase their recognition and early presentation.